Literature DB >> 23430834

Stroke and Stroke-Like Symptoms in Patients with Mutations in the POLG1 Gene.

Waleed Brinjikji1, Jerry W Swanson, Carrie Zabel, Peter J Dyck, Jennifer A Tracy, Ralitza H Gavrilova.   

Abstract

Introduction/Methods Mutations in POLG1, the gene encoding mitochondrial polymerase gamma (Polγ), have been associated with a number of well-characterized phenotypes. In this study, we report two cases of patients with biallelic POLG1 mutations and stroke. We also performed a review of the literature and report on all clinical studies of patients with POLG1 mutations in which stroke was described in the phenotype. For each patient, genotype and phenotype are reported. Results Including our two patients, a total of 22 patients have been reported with POLG1 mutations and stroke. The average age of onset of stroke in these patients was 9 years with a range of 1-23 years. In cases where localization was reported, the occipital lobes were the primary location of the infarct. Mutations in the linker-linker or linker-polymerase domains were the most frequent genotype observed. Seizures (16/22) and hepatic dysfunction/failure (8/22) were the most commonly reported symptoms in the stroke cohort. Conclusion This article raises an underrecognized point that patients with POLG1 mutations may suffer a cerebrovascular accident at a young age. The most common location of the infarction is in the occipital lobe. The presentation may be similar to MELAS and can be misdiagnosed as a migrainous stroke.

Entities:  

Year:  2011        PMID: 23430834      PMCID: PMC3509824          DOI: 10.1007/8904_2011_22

Source DB:  PubMed          Journal:  JIMD Rep        ISSN: 2192-8304


  23 in total

1.  Effects of L-arginine on the acute phase of strokes in three patients with MELAS.

Authors:  Y Koga; M Ishibashi; I Ueki; S Yatsuga; R Fukiyama; Y Akita; T Matsuishi
Journal:  Neurology       Date:  2002-03-12       Impact factor: 9.910

2.  POLG mutations associated with Alpers' syndrome and mitochondrial DNA depletion.

Authors:  Robert K Naviaux; Khue V Nguyen
Journal:  Ann Neurol       Date:  2004-05       Impact factor: 10.422

3.  Mutation of POLG is associated with progressive external ophthalmoplegia characterized by mtDNA deletions.

Authors:  G Van Goethem; B Dermaut; A Löfgren; J J Martin; C Van Broeckhoven
Journal:  Nat Genet       Date:  2001-07       Impact factor: 38.330

4.  Mitochondrial DNA polymerase gamma deficiency and mtDNA depletion in a child with Alpers' syndrome.

Authors:  R K Naviaux; W L Nyhan; B A Barshop; J Poulton; D Markusic; N C Karpinski; R H Haas
Journal:  Ann Neurol       Date:  1999-01       Impact factor: 10.422

5.  L-arginine improves the symptoms of strokelike episodes in MELAS.

Authors:  Y Koga; Y Akita; J Nishioka; S Yatsuga; N Povalko; Y Tanabe; S Fujimoto; Toyojiro Matsuishi
Journal:  Neurology       Date:  2005-02-22       Impact factor: 9.910

6.  Mono-allelic POLG expression resulting from nonsense-mediated decay and alternative splicing in a patient with Alpers syndrome.

Authors:  Sherine S L Chan; Matthew J Longley; Robert K Naviaux; William C Copeland
Journal:  DNA Repair (Amst)       Date:  2005-09-21

7.  Endothelial dysfunction in MELAS improved by l-arginine supplementation.

Authors:  Y Koga; Y Akita; N Junko; S Yatsuga; N Povalko; R Fukiyama; M Ishii; T Matsuishi
Journal:  Neurology       Date:  2006-06-13       Impact factor: 9.910

8.  The common A467T mutation in the human mitochondrial DNA polymerase (POLG) compromises catalytic efficiency and interaction with the accessory subunit.

Authors:  Sherine S L Chan; Matthew J Longley; William C Copeland
Journal:  J Biol Chem       Date:  2005-07-16       Impact factor: 5.157

9.  Functional defects due to spacer-region mutations of human mitochondrial DNA polymerase in a family with an ataxia-myopathy syndrome.

Authors:  Petri T Luoma; Ningguang Luo; Wolfgang N Löscher; Carol L Farr; Rita Horvath; Julia Wanschitz; Stefan Kiechl; Laurie S Kaguni; Anu Suomalainen
Journal:  Hum Mol Genet       Date:  2005-05-25       Impact factor: 6.150

10.  Alpers' syndrome presenting with seizures and multiple stroke-like episodes in a 17-year-old male.

Authors:  T J Montine; J M Powers; F S Vogel; R A Radtke
Journal:  Clin Neuropathol       Date:  1995 Nov-Dec       Impact factor: 1.368

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  2 in total

1.  Metabolic stroke in a patient with bi-allelic OPA1 mutations.

Authors:  Ayelet Zerem; Keren Yosovich; Yael Cohen Rappaport; Stephanie Libzon; Lubov Blumkin; Liat Ben-Sira; Dorit Lev; Tally Lerman-Sagie
Journal:  Metab Brain Dis       Date:  2019-04-10       Impact factor: 3.584

2.  Extensive respiratory chain defects in inhibitory interneurones in patients with mitochondrial disease.

Authors:  Nichola Z Lax; John Grady; Alex Laude; Felix Chan; Philippa D Hepplewhite; Grainne Gorman; Roger G Whittaker; Yi Ng; Mark O Cunningham; Doug M Turnbull
Journal:  Neuropathol Appl Neurobiol       Date:  2015-05-30       Impact factor: 8.090

  2 in total

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